Pleomorphic Adenoma Report of James D.
a
Leon
Barnes, MD; Vaughn B. Nixon, MD
\s=b\ We describe the clinical and pathologic aspects of an unusual case of pleomorphic adenoma of the epiglottis. A 69\x=req-\ year-old man had impaired speech and a "lumpy sensation" in the throat. Following clinical evaluation and a diagnostic biopsy, the tumor was totally excised with
excellent results. Pleomorphic adenoma of the larynx is most uncommon. To our knowledge, no report describing the clinical and pathologic features of this entity in the epiglottis or larynx has been previously reported. This is the only example of an epiglottic pleomorphic adenoma among 391 cases seen at Presbyterian-University Hospital and the Eye and Ear Hospital of Pittsburgh during a 21-year period.
(Arch Otolaryngol 103:245-247, 1977) adenomas
histo¬
Pleomorphi logically cbenign neoplasms hith¬ "benign are
mixed tumors."
pleomorphic adenomas arise in major salivary glands. However, their development in the minor salivary glands of the oral cavity, pharynx, paranasal sinuses, and in skin and Most
Accepted
for publication Dec 2, 1976. From the Departments of Pathology (Drs Cotelingam and Barnes) and Otolaryngology (Dr Nixon), Presbyterian-University Hospital, and Eye and Ear Hospital of Pittsburgh, University
Pittsburgh School of Medicine. Reprint requests to Department of Pathology, Presbyterian-University Hospital, 230 Lothrop St, Pittsburgh, PA 15213 (Dr Barnes).
of
Epiglottis
Case
Cotelingam, MD;
erto called
of the
lacrimal glands is well known. Despite the abundance of minor salivary gland type tissue in the larynx, pleomorphic adenoma is rare in this location.
3.5 cm oval, smooth, wellx 2.1 circumscribed, tan, and sessile tumor arising from the laryngeal aspect of the epiglottis (Fig 2). The lesion was 3.7
by a delicate connective tissue capsule. Semifirm pearly white and myxoid areas were present. The microscopic features of the lesion in the initial biopsy material and surgi¬ cal specimen were similar (Fig 3). The cellular component consisted of welldifferentiated clumps, and trabeculae of epithelial cells with focal duct formation and intervening myxoid and pseudocartilaginous matrix. The epithelial cells had centrally placed round-oval and at places spindleshaped nuclei with homogenous pale chromatin. Mitoses were not obvious. The cytoplasm of these cells was finely granular and deeply eosinophilic. No keratin or intercellular invested
REPORT OF A CASE A 69-year-old man came to PresbyterianUniversity Hospital in June 1975 with a one-week history of a "lumpy sensation" in his throat. He consumed alcohol regularly, and had discontinued pipe and cigarette smoking several years ago. On examination, he was well developed, in no acute distress, and had a character¬ istic "hot potato speech." Laryngoscopy revealed a large mass on the laryngeal side of the epiglottis with a ball valve effect. The vocal cords could not be visualized. No adenopathy was present, and no tumor was found elsewhere. A lateral x-ray view of the neck (Fig 1) disclosed a large well-circumscribed densi¬ ty about 3 cm in diameter, extending from the posteroinferior aspect of the epiglottis, with considerable decrease in the airway. A diagnostic biopsy was performed, and a histologie diagnosis of pleomorphic adenoma was made. Subsequently, pharyngotomy and tracheostomy were performed with the patient under general anesthesia, and the tumor, along with a portion of the epiglottis, was removed. The postoperative course was satisfactory, and the patient has been well and without recurrence for over 15 months.
PATHOLOGIC EXAMINATION The excised surgical specimen was a
bridges
were
present. The myxoid
matrix contained occasional small stellate myoepithelial cells, was metachromatic with toluidine blue, weakly PAS-positive diastase-resistant, and Alcian blue-positive. Secretions with¬ in ducts were strongly PAS-positive. COMMENT A wide variety of tumors arise in the larynx. These include carci¬ nomas,'-' papillomas,' polyps and
nodules,' amyloid tumors,'1 oncocytic cystadenomas," verrucous carcinoma,7
Downloaded From: http://archotol.jamanetwork.com/ by a Monash University Library User on 06/19/2015
Fig
1 .—Lateral x-ray view of neck. Tumor
(arrows)
is
supraglottic,
smooth, well circumscribed, and of soft-tissue density.
Fig 2.—Saggital cartilage.
section of
specimen: T, tumor; E, epiglottic
Distribution of Benign Adenoma*
Pleomorphic
Cases
Frequency, %
276
70.58
50
12.78
15 20 4 2
3.83 5.11 1.02 0.51 1.02
No. of
Location Parotid
glands Submandibular
glands salivary glands in buc¬
Minor
cal mucosa, floor of mouth,
and alveolar
ridge
Palate
Lips
Tonsillar
area
Pharynx
4
Paranasal
sinuses Nasal
cavity Epiglottis (present case)
Skin
External
Eyelids
ear
2 4 1
5 2 1
0.51 1.02 0.25 1.27 0.51 0.25
Lacrimal
glands
Total
5 391
"Presbyterian-University Hospital Hospital, 1955 to 1976.
1.27 and
Fig 3.—Characteristic microscopic appearance of pleomorphic adenoma. Note intact stratified squamous epithelium of epiglottis (hematoxylin-eosin, original magnification X40).
Eye and
Ear
Downloaded From: http://archotol.jamanetwork.com/ by a Monash University Library User on 06/19/2015
malignant melanoma,8 chemodecto¬ mas," plasmacytoma,"1 granular cell myoblastoma," rhabdomyosarcoma,12 neurofibroma,'3 angiomas,14 chondroma,
chondrosarcoma,' and metastatic '
carcinoma. A solitary example of laryngeal "mixed tumor" is cited by Cunning4 in a review of 1,335 laryngeal tumors from the Manhattan Eye and Ear Hospital. However, he does not de¬ scribe the location, morphologic fea¬ tures, or clinical aspects of this case. Several cases of "adenomata of the larynx" were published by Moore16 in 1920. His drawings illustrate a prolif¬ eration of glands and intervening stromal fat, and we are uncertain as to the exact nature of these cases. Large series on laryngeal tumors from the Mayo Clinic17 (722 cases) and University of Illinois'8 (1,197 cases) do not include an example of pleomorphic adenoma, and no case has been
from
our
files between 1955 and 1976
examined, and the data on 391 cases are presented in the Table. In this series, pleomorphic adenoma has occurred most frequently in the parot¬ id glands, and the present case was
represents the only example in the
larynx.
Studies relating to the histogenesis pleomorphic adenoma in salivary glands are controversial. However, in recent years, following histochemical and ultrastructural investigation, the role of the intercalated duct19 and myoepithelial cell2"-21 has been sus¬ pected. Our studies of control tissue have revealed that both intercalated ducts and myoepithelial cells can be of
previously diagnosed at PresbyterianUniversity and Eye and Ear Hospitals of Pittsburgh. Material on histiologically benign pleomorphic adenomas
clearly identified in the glandular complexes of the epiglottis. The relatively high frequency of parotid involvement remains an enig¬ ma. Implication of notochordal rests2'2 in the histogenesis is unlikely since it does not explain the pathogenesis in sites remote from the developing notochord. Although the comparative bulk of glandular tissue in the parotid
1. MaeComb WS: Cancer of the larynx. Cancer 19:149-156, 1966. 2. Randall G, Alonoso WA, Ogura JH: Spindle cell carcinoma (pseudosarcoma) of the larynx. Arch Otolaryngol 101:63-66, 1975. 3. Moore RL, Lattes R: Papillomatosis of larynx and bronchi. Cancer 12:117-126, 1959. 4. Cunning DS: Diagnosis and treatment of laryngeal tumors. JAMA 142:73-76 1950. 5. Leroux-Robert MJ: "Tumerus amyloides" du larynx. Ann Otolaryngol 79:249-270, 1962. 6. Kroe DJ, Pitcock JA, Cocke EW: Oncocytic papillary cystadenoma of the larynx. Arch Pathol 84:429-432, 1967. 7. Kraus FT, Perez-Mesa C: Verrucous carcinoma: Clinical and pathologic study of 105 cases involving oral cavity, larynx, and genitalia. Cancer 19:26-28, 1966. 8. Pentazopoulos PE: Primary malignant melanoma of the larynx. Laryngoscope 74:95-102, 1964.
9. Vetters JM, Toner PG: Chemodectoma of the larynx. J Pathol 101:259-265, 1970. 10. Costen JB: Plasmacytoma: A case with original lesion of the epiglottis and metastasis to the tibia. Laryngoscope 61:266-270, 1951. 11. Booth JB, Osborn DA: Granular cell myoblastoma of the larynx. Acta Otolaryngol 70:279-293, 1970. 12. Batsakis JG, Fox JE: Rhabdomyosarcoma of the larynx. Arch Otolaryngol 91:136-140, 1970. 13. Calvet J, Claux G: Le neurinome du larynx. Rev Laryngol Otol Rhinol 69:47-60, 1948. 14. Tefft M: The radiotherapeutic management of subglottic hemangioma in children. Radiology 85:207-214, 1966. 15. Goethals PL, Dahlin DC, Devine KD: Cartilaginous tumours of the larynx. Surg Gynecol Obstet 117:77-82, 1963. 16. Moore I: Adenomata (glandular tumours) of the larynx. J Laryngol 35:65-75, 1920.
may be important, the purely serous nature of this tissue could be relevant. In minor salivary glands, where the incidence is lower, mucinous in addi¬ tion to serous units are present. The serous: mucinous gland ratio is higher in submandibular glands and lower in sublingual glands.23 In the epiglottis, our control material reveals a uniform and equal distribution of these compo¬ nents. The morphologic appearance of this epiglottic tumor is quite characteris¬
tic, and its myxoid stromal component demonstrates the cytochemical fea¬ tures of connective tissue mucin."'
We hope that this case will serve to establish pleomorphic adenoma as a rare albeit definite entity in the differential diagnosis of laryngeal tumors.
The late Robert S. Totten, MD, made possible the accumulation of data on pleomorphic adeno¬ ma. Vincent Hyams, MD, reviewed this case. Donna Short provided technical assistance and Tish Clark assisted in the preparation of the
manuscript.
References 17. New GB, Erich JB: Benign tumours of the larynx. Arch Otolaryngol 28:841-910, 1938. 18. Holinger PH, Johnston KC: Benign tumors of the larynx. Ann Otol Rhinol Laryngol 60:496\x=req-\
509, 1951.
19. vary
Azzopardi JG, Smith OD: Mucins in saligland tumours. J Pathol Bacteriol 77:131\x=req-\
140, 1959.
20. H\l=u"\bnerG, Klein HJ, Leinsasser O, et al: Role of myoepithelial cells in the development of salivary gland tumours. Cancer 27:1255-1261, 1971. 21. Mylius EA: The identification and role of the myoepithelial cell in salivary gland tumors. Acta Pathol Microbiol Scand 50 (suppl 139):1-59, 1960. 22. Hellwig CA: Mixed tumors of the salivary glands. Arch Pathol 40:1-10, 1945. 23. Ham AW: The salivary glands, in Histology, ed 7. Philadelphia, JB Lippincot Co, Inc, 1974, p 645.
Downloaded From: http://archotol.jamanetwork.com/ by a Monash University Library User on 06/19/2015
a
Leon
Barnes, MD; Vaughn B. Nixon, MD
\s=b\ We describe the clinical and pathologic aspects of an unusual case of pleomorphic adenoma of the epiglottis. A 69\x=req-\ year-old man had impaired speech and a "lumpy sensation" in the throat. Following clinical evaluation and a diagnostic biopsy, the tumor was totally excised with
excellent results. Pleomorphic adenoma of the larynx is most uncommon. To our knowledge, no report describing the clinical and pathologic features of this entity in the epiglottis or larynx has been previously reported. This is the only example of an epiglottic pleomorphic adenoma among 391 cases seen at Presbyterian-University Hospital and the Eye and Ear Hospital of Pittsburgh during a 21-year period.
(Arch Otolaryngol 103:245-247, 1977) adenomas
histo¬
Pleomorphi logically cbenign neoplasms hith¬ "benign are
mixed tumors."
pleomorphic adenomas arise in major salivary glands. However, their development in the minor salivary glands of the oral cavity, pharynx, paranasal sinuses, and in skin and Most
Accepted
for publication Dec 2, 1976. From the Departments of Pathology (Drs Cotelingam and Barnes) and Otolaryngology (Dr Nixon), Presbyterian-University Hospital, and Eye and Ear Hospital of Pittsburgh, University
Pittsburgh School of Medicine. Reprint requests to Department of Pathology, Presbyterian-University Hospital, 230 Lothrop St, Pittsburgh, PA 15213 (Dr Barnes).
of
Epiglottis
Case
Cotelingam, MD;
erto called
of the
lacrimal glands is well known. Despite the abundance of minor salivary gland type tissue in the larynx, pleomorphic adenoma is rare in this location.
3.5 cm oval, smooth, wellx 2.1 circumscribed, tan, and sessile tumor arising from the laryngeal aspect of the epiglottis (Fig 2). The lesion was 3.7
by a delicate connective tissue capsule. Semifirm pearly white and myxoid areas were present. The microscopic features of the lesion in the initial biopsy material and surgi¬ cal specimen were similar (Fig 3). The cellular component consisted of welldifferentiated clumps, and trabeculae of epithelial cells with focal duct formation and intervening myxoid and pseudocartilaginous matrix. The epithelial cells had centrally placed round-oval and at places spindleshaped nuclei with homogenous pale chromatin. Mitoses were not obvious. The cytoplasm of these cells was finely granular and deeply eosinophilic. No keratin or intercellular invested
REPORT OF A CASE A 69-year-old man came to PresbyterianUniversity Hospital in June 1975 with a one-week history of a "lumpy sensation" in his throat. He consumed alcohol regularly, and had discontinued pipe and cigarette smoking several years ago. On examination, he was well developed, in no acute distress, and had a character¬ istic "hot potato speech." Laryngoscopy revealed a large mass on the laryngeal side of the epiglottis with a ball valve effect. The vocal cords could not be visualized. No adenopathy was present, and no tumor was found elsewhere. A lateral x-ray view of the neck (Fig 1) disclosed a large well-circumscribed densi¬ ty about 3 cm in diameter, extending from the posteroinferior aspect of the epiglottis, with considerable decrease in the airway. A diagnostic biopsy was performed, and a histologie diagnosis of pleomorphic adenoma was made. Subsequently, pharyngotomy and tracheostomy were performed with the patient under general anesthesia, and the tumor, along with a portion of the epiglottis, was removed. The postoperative course was satisfactory, and the patient has been well and without recurrence for over 15 months.
PATHOLOGIC EXAMINATION The excised surgical specimen was a
bridges
were
present. The myxoid
matrix contained occasional small stellate myoepithelial cells, was metachromatic with toluidine blue, weakly PAS-positive diastase-resistant, and Alcian blue-positive. Secretions with¬ in ducts were strongly PAS-positive. COMMENT A wide variety of tumors arise in the larynx. These include carci¬ nomas,'-' papillomas,' polyps and
nodules,' amyloid tumors,'1 oncocytic cystadenomas," verrucous carcinoma,7
Downloaded From: http://archotol.jamanetwork.com/ by a Monash University Library User on 06/19/2015
Fig
1 .—Lateral x-ray view of neck. Tumor
(arrows)
is
supraglottic,
smooth, well circumscribed, and of soft-tissue density.
Fig 2.—Saggital cartilage.
section of
specimen: T, tumor; E, epiglottic
Distribution of Benign Adenoma*
Pleomorphic
Cases
Frequency, %
276
70.58
50
12.78
15 20 4 2
3.83 5.11 1.02 0.51 1.02
No. of
Location Parotid
glands Submandibular
glands salivary glands in buc¬
Minor
cal mucosa, floor of mouth,
and alveolar
ridge
Palate
Lips
Tonsillar
area
Pharynx
4
Paranasal
sinuses Nasal
cavity Epiglottis (present case)
Skin
External
Eyelids
ear
2 4 1
5 2 1
0.51 1.02 0.25 1.27 0.51 0.25
Lacrimal
glands
Total
5 391
"Presbyterian-University Hospital Hospital, 1955 to 1976.
1.27 and
Fig 3.—Characteristic microscopic appearance of pleomorphic adenoma. Note intact stratified squamous epithelium of epiglottis (hematoxylin-eosin, original magnification X40).
Eye and
Ear
Downloaded From: http://archotol.jamanetwork.com/ by a Monash University Library User on 06/19/2015
malignant melanoma,8 chemodecto¬ mas," plasmacytoma,"1 granular cell myoblastoma," rhabdomyosarcoma,12 neurofibroma,'3 angiomas,14 chondroma,
chondrosarcoma,' and metastatic '
carcinoma. A solitary example of laryngeal "mixed tumor" is cited by Cunning4 in a review of 1,335 laryngeal tumors from the Manhattan Eye and Ear Hospital. However, he does not de¬ scribe the location, morphologic fea¬ tures, or clinical aspects of this case. Several cases of "adenomata of the larynx" were published by Moore16 in 1920. His drawings illustrate a prolif¬ eration of glands and intervening stromal fat, and we are uncertain as to the exact nature of these cases. Large series on laryngeal tumors from the Mayo Clinic17 (722 cases) and University of Illinois'8 (1,197 cases) do not include an example of pleomorphic adenoma, and no case has been
from
our
files between 1955 and 1976
examined, and the data on 391 cases are presented in the Table. In this series, pleomorphic adenoma has occurred most frequently in the parot¬ id glands, and the present case was
represents the only example in the
larynx.
Studies relating to the histogenesis pleomorphic adenoma in salivary glands are controversial. However, in recent years, following histochemical and ultrastructural investigation, the role of the intercalated duct19 and myoepithelial cell2"-21 has been sus¬ pected. Our studies of control tissue have revealed that both intercalated ducts and myoepithelial cells can be of
previously diagnosed at PresbyterianUniversity and Eye and Ear Hospitals of Pittsburgh. Material on histiologically benign pleomorphic adenomas
clearly identified in the glandular complexes of the epiglottis. The relatively high frequency of parotid involvement remains an enig¬ ma. Implication of notochordal rests2'2 in the histogenesis is unlikely since it does not explain the pathogenesis in sites remote from the developing notochord. Although the comparative bulk of glandular tissue in the parotid
1. MaeComb WS: Cancer of the larynx. Cancer 19:149-156, 1966. 2. Randall G, Alonoso WA, Ogura JH: Spindle cell carcinoma (pseudosarcoma) of the larynx. Arch Otolaryngol 101:63-66, 1975. 3. Moore RL, Lattes R: Papillomatosis of larynx and bronchi. Cancer 12:117-126, 1959. 4. Cunning DS: Diagnosis and treatment of laryngeal tumors. JAMA 142:73-76 1950. 5. Leroux-Robert MJ: "Tumerus amyloides" du larynx. Ann Otolaryngol 79:249-270, 1962. 6. Kroe DJ, Pitcock JA, Cocke EW: Oncocytic papillary cystadenoma of the larynx. Arch Pathol 84:429-432, 1967. 7. Kraus FT, Perez-Mesa C: Verrucous carcinoma: Clinical and pathologic study of 105 cases involving oral cavity, larynx, and genitalia. Cancer 19:26-28, 1966. 8. Pentazopoulos PE: Primary malignant melanoma of the larynx. Laryngoscope 74:95-102, 1964.
9. Vetters JM, Toner PG: Chemodectoma of the larynx. J Pathol 101:259-265, 1970. 10. Costen JB: Plasmacytoma: A case with original lesion of the epiglottis and metastasis to the tibia. Laryngoscope 61:266-270, 1951. 11. Booth JB, Osborn DA: Granular cell myoblastoma of the larynx. Acta Otolaryngol 70:279-293, 1970. 12. Batsakis JG, Fox JE: Rhabdomyosarcoma of the larynx. Arch Otolaryngol 91:136-140, 1970. 13. Calvet J, Claux G: Le neurinome du larynx. Rev Laryngol Otol Rhinol 69:47-60, 1948. 14. Tefft M: The radiotherapeutic management of subglottic hemangioma in children. Radiology 85:207-214, 1966. 15. Goethals PL, Dahlin DC, Devine KD: Cartilaginous tumours of the larynx. Surg Gynecol Obstet 117:77-82, 1963. 16. Moore I: Adenomata (glandular tumours) of the larynx. J Laryngol 35:65-75, 1920.
may be important, the purely serous nature of this tissue could be relevant. In minor salivary glands, where the incidence is lower, mucinous in addi¬ tion to serous units are present. The serous: mucinous gland ratio is higher in submandibular glands and lower in sublingual glands.23 In the epiglottis, our control material reveals a uniform and equal distribution of these compo¬ nents. The morphologic appearance of this epiglottic tumor is quite characteris¬
tic, and its myxoid stromal component demonstrates the cytochemical fea¬ tures of connective tissue mucin."'
We hope that this case will serve to establish pleomorphic adenoma as a rare albeit definite entity in the differential diagnosis of laryngeal tumors.
The late Robert S. Totten, MD, made possible the accumulation of data on pleomorphic adeno¬ ma. Vincent Hyams, MD, reviewed this case. Donna Short provided technical assistance and Tish Clark assisted in the preparation of the
manuscript.
References 17. New GB, Erich JB: Benign tumours of the larynx. Arch Otolaryngol 28:841-910, 1938. 18. Holinger PH, Johnston KC: Benign tumors of the larynx. Ann Otol Rhinol Laryngol 60:496\x=req-\
509, 1951.
19. vary
Azzopardi JG, Smith OD: Mucins in saligland tumours. J Pathol Bacteriol 77:131\x=req-\
140, 1959.
20. H\l=u"\bnerG, Klein HJ, Leinsasser O, et al: Role of myoepithelial cells in the development of salivary gland tumours. Cancer 27:1255-1261, 1971. 21. Mylius EA: The identification and role of the myoepithelial cell in salivary gland tumors. Acta Pathol Microbiol Scand 50 (suppl 139):1-59, 1960. 22. Hellwig CA: Mixed tumors of the salivary glands. Arch Pathol 40:1-10, 1945. 23. Ham AW: The salivary glands, in Histology, ed 7. Philadelphia, JB Lippincot Co, Inc, 1974, p 645.
Downloaded From: http://archotol.jamanetwork.com/ by a Monash University Library User on 06/19/2015
